Sir,
A 51-year-old male presented with erythematous to brownish ill-defined indurated enlarging plaques on the right leg, left flank, and left anterior chest over 10 days with hardness. All of the lesions were exceeding 5 cm in diameter [Figures 1 and 2]. He did not give tick bite history and never had traveled to the endemic areas of Lyme borreliosis. Biopsy from the right leg showed lymphoplasmacytic infiltrate and fibrosis in the dermis and subcutis [Figures 3 and 4]. Complete blood count, chemistry, HIV testing were normal, and antinuclear antibody, anti-Ro/La, anti-RNP, anti Scl70, anti-centromere antibody were all negative. Lyme disease immunoglobulin G titer was 1:512 and IgM titer was <1:16 in immunofluorescence assay (IFA), but negative in Western blot test. Warthin Starry stain was negative. DNA was not detected in polymerase chain reaction.
Figure 1.
Erythematous to brown colored, irregularly circumscribed indurated plaques are found from upper thigh to calf
Figure 2.
Slightly whitish to erythematous irregular shaped indurated patch on chest, around nipple
Figure 3.
Histopathology of specimen from right calf shows perivascular and interstitial infiltrates of lymphocytes and plasma cells in dermis (H and E, ×40)
Figure 4.
Patchy fibrosis in the subcutaneous layer, and lymphoplasmacytic infiltrates are found. Trabeculae are thickened in subcutaneous layer (H and E, ×100)
Transthoracic echocardiography, electrocardiography, esophagogastroduodenoscopy, colonofibroscopy, chest and abdominopelvic computed tomography, and pulmonary function test were done and all were normal. Oral doxycycline (200 mg/day) for 3 weeks was started due to the possibility of Lyme borreliosis but without response. After systemic corticosteroid administration, the progression was slowed. After discontinuing the medication for 3 months, new skin lesions were found on the left buttock and thigh. Corticosteroid was started again, and the progression was stopped. There was no progression after cessation of medication until reporting.
Follow-up IFA and western blot determinations were performed 6 months after the onset of symptom, but the titer did not decrease, and western blot was still negative.
Lyme disease, also known as Lyme borreliosis, is a multi-organ infection caused by spirochetes of the Borrelia burgdorferi sensu lato group which are transmitted by ticks of the species Ixodes.[1] Common skin manifestations include erythema migrans, lymphocytoma, and acrodermatitis chronica atrophicans.[1] Recently, there have been some reports linking morphea or lichen sclerosus to B. burgdorferi infection.[1]
The presence of antibodies to B. burgdorferi in 50% of patients with morphea was found with enzyme-linked immunosorbent assay (ELISA),[1] but contradictory data have been proposed.[2] Most reports of the association between positive antibody test to B. burgdorferi and morphea come from Europe, and have been rarely found in the USA.[3] Differences between Borrelia strains and prevalence in the USA and in Europe have been raised to explain these conflicting results.[3] B. garinii, B. afzelii, and B. burgdorferi sensu stricto are present in Europe, only B. burgdorferi sensu stricto is identified in the USA. It has been suggested that only certain types of infection of B. burgdorferi may lead to morphea. B. burgdorferi sensu stricto, has never been found in late dermatologic manifestations of Lyme borreliosis, this may explain the reason of conflicting data from different regions.[3]
The overall false-positive rate of Lyme borreliosis testing is approximately 5%.[4] When patients have other viral or spirochetal infections or autoimmune diseases, false-positive findings with ELISA or IFA may occur.[4] There has been only one case of morphea with false positivity to Lyme titer since 1993.[5] We experienced this interesting case of rapidly progressing generalized morphea with high antibody titer to B. burgdorferi.
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The authors certify that they have obtained all appropriate patient consent forms. In the form the patient(s) has/have given his/her/their consent for his/her/their images and other clinical information to be reported in the journal. The patients understand that their names and initials will not be published and due efforts will be made to conceal their identity, but anonymity cannot be guaranteed.
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Conflicts of interest
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References
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